% IMPORTANT: The following is UTF-8 encoded. This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.
@INPROCEEDINGS{Pasternak:283071,
author = {Pasternak, Maurice and Mirza, Saira S and Paterson, Andrew
D and Tartaglia, Carmela and Mitchell, Sara and Black,
Sandra E and Freedman, Morris and Tang-Wai, David F and
Rogaeva, Ekaterina and Cash, David M and Bocchetta, Martina
and van Swieten, John and Laforce, Robert and Tagliavini,
Fabrizio and Borroni, Barbara and Galimberti, Daniela and
Rowe, James B and Graff, Caroline and Finger, Elizabeth and
Sorbi, Sandro and de Mendonça, Alexandre and Butler,
Christopher and Gerhard, Alexander and Sánchez-Valle,
Raquel and Moreno, Fermin and Synofzik, Matthis and
Vandenberghe, Rik and Ducharme, Simon and Levin, Johannes
and Otto, Markus and Santana, Isabel and Rohrer, Jonathan D
and Masellis, Mario},
collaboration = {GENFI},
title = {{TMEM}106{B} modulates disease severity in genetic
frontotemporal dementia phenoconverters},
journal = {Alzheimer's and dementia},
volume = {21 Suppl 2},
number = {Suppl 2},
issn = {1552-5260},
reportid = {DZNE-2025-01478},
pages = {e103532},
year = {2025},
abstract = {A common variant within TMEM106B is associated with risk
for Frontotemporal Lobar Degeneration-Tar DNA binding
Protein-43 (FTLD-TDP). A recent study has shown that the
minor allele G of TMEM106B-rs1990622 confers protection
against FTLD-TDP in symptomatic mutation carriers through
reductions in NfL serum levels, brain atrophy, and cognitive
decline. It is unknown whether this protective effect is
present in phenoconverters of the disease.We included 518
participants from the GENetic Frontotemporal dementia
Initiative (GENFI), which recruits genetic FTD cases and
their family members, both carriers and non-carriers of FTD
mutations. Of these, 21 were phenoconverters, 209 were
non-carrier controls, 70 were presymptomatic and 45
symptomatic C9orf72 carriers, 92 presymptomatic and 29
symptomatic GRN carriers, and 39 presymptomatic and 13
symptomatic MAPT carriers. Effects of interaction between
TMEM106B-rs1990622 and phenoconverter status were examined
using mixed effects models, with a random effects structure
featuring subjects nested within families and fixed effects
for age at baseline and sex. Serum neurofilament light chain
(NfL) was measured using the Simoa platform. Cognitive
assessment included the Mini-Mental State Examination
(MMSE), tests of attention, processing speed, executive
function, and language, as well as the Cambridge Behavioural
Inventory (CBI), with mixed effects also including years of
education as a covariate. Brain volumetry was assessed using
T1-weighted MRI and these mixed effect models also included
additional covariates of total intracranial volume and
scanner site.In phenoconverters, each copy of the protective
allele G was associated with a significant reduction in the
rate of serum NfL accumulation (-5.33 pg/mL/year; p = 7.79
× 10-9). Structural imaging analyses revealed decreased
rates of atrophy in fronto-orbital regions and the insular
cortex among protective allele carriers. Cognitive
trajectories showed significantly slower decline across
multiple domains including general cognition (MMSE; p =
0.003), attention and processing speed (p = 2.2 × 10-4),
executive function (p = 2.6 × 10-7), language (p = 2.9 ×
10-3), and behavioural symptoms as measured by CBI (p = 9.5
× 10-3).The TMEM106B-rs1990622 protective variant
significantly modulates disease progression in genetic FTD
phenoconverters across multiple markers, suggesting its
potential as a therapeutic target.},
month = {Jul},
date = {2025-07-27},
organization = {Alzheimer’s Association
International Conference, Toronto
(Canada), 27 Jul 2025 - 31 Jul 2025},
keywords = {Humans / Male / Female / Nerve Tissue Proteins: genetics /
Middle Aged / Biomarkers: blood / Membrane Proteins:
genetics / Frontotemporal Dementia: genetics /
Frontotemporal Dementia: pathology / Aged / C9orf72 Protein:
genetics / Brain: pathology / Brain: diagnostic imaging /
tau Proteins: genetics / Progranulins: genetics /
Neurofilament Proteins: blood / Mutation: genetics /
Magnetic Resonance Imaging / Atrophy / Nerve Tissue Proteins
(NLM Chemicals) / Biomarkers (NLM Chemicals) / TMEM106B
protein, human (NLM Chemicals) / Membrane Proteins (NLM
Chemicals) / C9orf72 Protein (NLM Chemicals) / tau Proteins
(NLM Chemicals) / Progranulins (NLM Chemicals) /
Neurofilament Proteins (NLM Chemicals) / C9orf72 protein,
human (NLM Chemicals) / MAPT protein, human (NLM Chemicals)
/ neurofilament protein L (NLM Chemicals)},
cin = {AG Gasser / Clinical Research (Munich) / AG Levin},
ddc = {610},
cid = {I:(DE-2719)1210000 / I:(DE-2719)1111015 /
I:(DE-2719)1111016},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)1 / PUB:(DE-HGF)16},
pubmed = {pmid:41449512},
pmc = {pmc:PMC12741048},
doi = {10.1002/alz70856_103532},
url = {https://pub.dzne.de/record/283071},
}